Cold Spring Harbor molecular case studies · 2022
Whole-exome sequencing detects PYGM variants in two adults with McArdle disease.
СтатьяНазвание и аннотация: Перевод в очереди
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Thomas-Wilson Amanda, Dharmadhikari Avinash V, Heymann Jonas J, Jobanputra Vaidehi, DiMauro Salvatore, Hirano Michio, Naini Ali B, Ganapathi Mythily
Аннотация
McArdle disease is a debilitating glycogen storage disease with typical onset in childhood. Here, we describe a former competitive athlete with early adult-onset McArdle disease and a septuagenarian with a history of exercise intolerance since adolescence who was evaluated for proximal muscle weakness. Exome sequencing identified biallelic variants in the PYGM gene for both cases. The former athlete has the common, well-known pathogenic variant p.(Arg50Ter) in trans with a novel missense variant, p.(Asp694Glu). The second individual has a previously described homozygous missense variant, p.(Arg771Gln). Here, we describe the clinical course, enzyme-testing results using muscle tissue, and molecular findings for the individuals and add to the knowledge of the genotypic spectrum of this disorder.